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      A Case of Loose Anagen Hair Syndrome in a Southeast Asian Boy

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          Abstract

          Loose anagen hair syndrome is an uncommon hair disorder, particularly in non-Caucasian children. We report the case of a 13-year-old Thai boy who presented with a single patch of hair thinning on the frontal scalp with excessive shedding, and the hairs did not grow long. Microscopic examination showed naked anagen bulbs with ruffling of the cuticle, which is compatible with loose anagen hair syndrome. To our knowledge, there is no reported case in Southeast Asian children.

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          Most cited references17

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          Loose anagen hair syndrome and loose anagen hair.

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            Is the loose anagen hair syndrome a keratin disorder? A clinical and molecular study.

            To report the clinical features of the loose anagen hair syndrome and to test the hypothesis that the typical gap between the hair and the inner root sheath may result from hereditary defects in the inner root sheath or the apposed companion layer. Case series. A pediatric dermatology unit (referral center). A consecutive sample of 17 children (13 girls). For 9 of them and their first-degree relatives, molecular analyses were performed in the K6HF gene with 50 appropriate controls. Minoxidil therapy (5% lotion) in 11 patients for 1 to 12 months. Clinical and follow-up features and determination of mutations in the K6HF gene. Most patients had easily pluckable hair with no sign of scalp inflammation or scarring. Ten patients seldom cut their hair, and 4 had unmanageable hair. One patient had hypodontia. Two patients had an additional clinical phenotype of diffuse partial woolly hair. The family history was positive for loose anagen hair syndrome in 5 patients. Marked improvement was noted after treatment with 5% minoxidil lotion in 7 of the 11 patients treated. Polymerase chain reaction analysis of the gene segments encoding the alpha-helical 1A and 2B subdomains of K6hf, the type II cytokeratin exclusively expressed in the companion layer, was performed in 9 families. In 3 of these 9 families, a heterozygous glutamic acid and lysine mutation, E337K, was identified in the L2 linker region of K6HF. Diffuse partial woolly hair can be associated with loose anagen hair syndrome. A keratin mutation, E337K in K6HF, was possibly causative in 3 of the 9 families studied. Another keratin, and possibly the type I partner of K6hf, could be responsible for loose anagen hair syndrome in other patients, or the gene involved may be a minor gene.
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              Loose anagen syndrome.

              A distinctive new hair condition, the loose anagen syndrome, features anagen hairs that are loosely anchored and easily pulled from the scalp. The children studied had sparse hair that did not grow long and that pulled easily from the scalp. The majority of patients were blond girls, aged 2 to 5 years, but both sexes and those with dark hair can be affected. The hair was not fragile, and easily pulled hairs were misshapen anagen hairs without external root sheaths. Histologic examination of the hair showed abnormal premature keratinization of Huxley's and Henle's layers of the inner root sheath in some samples. Length and density of hair gradually increased with age, but anagen hairs remained loosely anchored in adulthood. This report describes findings in 22 children and five adults with the loose anagen syndrome.
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                Author and article information

                Journal
                CDE
                CDE
                10.1159/issn.1662-6567
                Case Reports in Dermatology
                S. Karger AG
                1662-6567
                2019
                May -August 2019
                03 July 2019
                : 11
                : 2
                : 204-208
                Affiliations
                Division of Dermatology, Faculty of Medicine, Ramathibodi Hospital, Mahidol University, Bangkok, Thailand
                Author notes
                *Poonkiat Suchonwanit, MD, Division of Dermatology, Faculty of Medicine, Ramathibodi Hospital, Mahidol University, 270 Rama VI Road, Rajthevi, Bangkok 10400 (Thailand), E-Mail poonkiat@hotmail.com
                Author information
                https://orcid.org/0000-0001-9723-0563
                Article
                501443 PMC6639585 Case Rep Dermatol 2019;11:204–208
                10.1159/000501443
                PMC6639585
                31341463
                b205a6b9-f311-472d-afa7-38ae52e392f3
                © 2019 The Author(s). Published by S. Karger AG, Basel

                This article is licensed under the Creative Commons Attribution-NonCommercial 4.0 International License (CC BY-NC). Usage and distribution for commercial purposes requires written permission. Drug Dosage: The authors and the publisher have exerted every effort to ensure that drug selection and dosage set forth in this text are in accord with current recommendations and practice at the time of publication. However, in view of ongoing research, changes in government regulations, and the constant flow of information relating to drug therapy and drug reactions, the reader is urged to check the package insert for each drug for any changes in indications and dosage and for added warnings and precautions. This is particularly important when the recommended agent is a new and/or infrequently employed drug. Disclaimer: The statements, opinions and data contained in this publication are solely those of the individual authors and contributors and not of the publishers and the editor(s). The appearance of advertisements or/and product references in the publication is not a warranty, endorsement, or approval of the products or services advertised or of their effectiveness, quality or safety. The publisher and the editor(s) disclaim responsibility for any injury to persons or property resulting from any ideas, methods, instructions or products referred to in the content or advertisements.

                History
                : 20 May 2019
                : 12 June 2019
                Page count
                Figures: 2, Pages: 5
                Categories
                Case and Review

                Oncology & Radiotherapy,Pathology,Surgery,Dermatology,Pharmacology & Pharmaceutical medicine
                Hair loss,Alopecia,Ruffling cuticle,Floppy sock appearance,Pluckable hair

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